Case ReportSpontaneous Hemopneumothorax: A Rare Cause of UnexplainedHemodynamic Instability in a Young Patient

Antonio Higor Marques Aragão ,1 Letícia Aguiar Fonseca,1 Flávio Clemente Deulefeu,2

Israel Lopes Medeiros ,3 Rafael Fernandes Viana de Araújo,3 Carlos Alberto da Cruz Neto,3

and Antero Gomes Neto3

1College of Medicine, Universidade Estadual do Ceará, Fortaleza, CE, Brazil2Department of Pulmonology, Hospital de Messejana Dr. Carlos Alberto Studart Gomes, Fortaleza, CE, Brazil3Department of Thoracic Surgery, Hospital de Messejana Dr. Carlos Alberto Studart Gomes, Fortaleza, CE, Brazil

Correspondence should be addressed to Israel Lopes Medeiros; israelmed@globo.com

Received 11 May 2019; Accepted 4 October 2019; Published 25 January 2020

Academic Editor: Akif Turna

Copyright © 2020 Antonio Higor Marques Aragão et al. This is an open access article distributed under the Creative CommonsAttribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original workis properly cited.

Spontaneous hemopneumothorax is a rare and potentially life-threatening disorder which complicates about 1-12% of patientspresenting with spontaneous pneumothorax and has a remarkable predilection for male patients. It may present with signs ofhypovolemic shock without apparent cause. While there are no specific guidelines for the management of patients diagnosedwith such condition, wide debate in the literature relating to patient selection for surgery remains unresolved, and recently thereseems to be a trend increasingly favorable towards early surgical intervention. Video-assisted thoracic surgery emerges as anexcellent option for stable patients and has now been considered the gold standard treatment for spontaneoushemopneumothorax. We report the case of a 17-year-old male patient who presented to the emergency department with ahistory of sudden chest pain and dyspnea, with no previous evidence of trauma. On admission, the patient presented withhypotension, tachycardia, and cutaneous pallor. Chest X-ray showed hydropneumothorax on the left hemithorax; then, chesttube was placed with an initial drainage of 2000ml of blood.

1. Introduction

Over the past one and a half century, spontaneous hemopneu-mothorax (SHP) has emerged as a rare well-documentedentity in the literature, being first described by Laënnec in1828, and it complicates about 1-12% of all spontaneouspneumothoraces. Although uncommon, SHP is a potentiallylife-threatening condition and, therefore, must be remem-bered in the context of sudden onset of unexplained hemody-namic instability, especially in young males, since proper andimmediate management in these cases is a determining factorfor both treatment success and to improve prognosis [1].

Although the role of different surgical modalities in theapproach to SHP has gained increasing importance over thelast few years, the literature still diverges about its indication,

the ideal moment for its execution, and the possibility of iso-lated conservative treatment in selected cases [2–5].

We describe the case of a 17-year-old patient who pre-sented with sudden onset of respiratory distress and signsof hypovolemia and discuss the current state-of-the-art ofsuch condition.

2. Case Presentation

A 17-year-old male patient presents to the emergency depart-ment with a two-hour history of sudden onset of left-sidedchest pain, exacerbated by breathing, with no radiation toother sites, and associated with shortness of breath. Thepatient reported no recent trauma and his past medical historywas unremarkable. On admission, his vital signs revealed

HindawiCase Reports in PulmonologyVolume 2020, Article ID 5026759, 5 pageshttps://doi.org/10.1155/2020/5026759

temperature of 34.5°C, blood pressure of 80/40mmHg, heartrate of 125 beats/min, and respiratory rate of 24 breaths/minwith an oxygen saturation of 98% on room air. At phys-ical examination, the patient was dyspneic and presentedimportant painful distress. Cardiopulmonary examinationwas significant for muffled heart sounds on mitral focusand abolished breathing sounds in the left hemithorax.Electrocardiography (ECG) showed normal sinus rhythm,and initial laboratory results were notable only for a hemo-globin level of 8mg/dl. Chest X-ray (CXR) evidenced a left-sided hydropneumothorax (Figure 1).

A computed tomography (CT) scan of the chest revealedleft lung collapse with significant deviation of the mediasti-num to the right side (Figure 2). A thoracentesis confirmedthe presence of blood in the pleural cavity. A chest tubewas then inserted in the 5th intercostal space with drainageof air and 2000ml of blood. Two units of packed red bloodcells were administered and the patient’s vital signs stabi-lized as well as his symptoms relieved. The patient wasreferred to a tertiary cardiopulmonary service for definitivetreatment evaluation.

Within the next hours till presentation to our service,additional 800ml of blood were drained. New CXR revealedadequate lung reexpansion after tube insertion (Figure 3),and the thoracic surgery team decided not to proceed toemergency thoracotomy. The patient was admitted, and overthe next 24 hours, another 200ml of blood flowed out fromthe chest tube. One more unit of packed red blood cells wastransfused and signs of hemodynamic instability did notreoccur during hospitalization. Owing to the persistentbleeding, the medical team chose to indicate video-assistedthoracic surgery (VATS) to accomplish definitive hemostasisof the bleeding source.

On the third hospitalization day, VATS was performed,after induction of general anesthesia, with camera insertedthrough the already existing thoracostomy tube incisionand confection of a portal in the 4th intercostal space. Explo-ration of the pleural cavity revealed a large amount ofretained blood clot with pleural adhesion exhibiting signs ofrecent bleeding. A small bullae was found in the apex of theleft upper lobe, near the site where a pleural adhesion rup-tured (Figures 4 and 5). Complete removal of clots, irrigationof the pleural cavity, electrocauterization of the bleedingadhesion, bullectomy, and abrasive pleurodesis were effectu-ated. A new port was made in the 7th intercostal space forchest tube insertion and the previous one was closed.

Histopathology showed reactive eosinophilic pleuritisconsistent with previous pneumothorax, and no morpholog-ical evidence of malignancy or granulomas was present.

The postoperative course was uneventful, and the chesttube was removed on the 7th postoperative day. The patientremained stable and was discharged on the 10th postopera-tive day.

Seven days after hospital discharge, the patient presentsasymptomatic to our service for an outpatient follow-upappointment. However, CXR showed radiologic signs ofpneumothorax on the right hemithorax, which was then con-firmed by a CT scan of the chest (Figure 6). The patientunderwent new thoracoscopy with bullectomy and pleurod-esis and was discharged 2 days after the procedure. Thepatient continued to be reevaluated and no more complica-tions were observed at a 2-month follow-up.

3. Discussion

The definition of SHT is not yet unanimous in the litera-ture. Some authors adopt the concept proposed by Ohmoriin 1988 as the accumulation of more than 400ml of bloodin the pleural cavity associated with primary spontaneouspneumothorax (PSP) [1, 4, 6]. On the other hand, a recentmeta-analysis proposed that any hemothorax accompany-ing spontaneous pneumothorax is a more reasonable defi-nition for this condition once there are cases reported inthe literature in which the initial drainage was less than400ml [2, 7].

Almost all described cases of SHT occurred as a compli-cation of a primary spontaneous pneumothorax (PSP). Theincidence of SHT has been reported to be around 1-12% ofall PSP. A notorious predisposition for male patients is welldocumented, as the male to female ratio range is approxi-mately 15 : 1, difference significantly higher compared toPSP [1, 7, 8].

The most frequent presenting symptoms are chest painand sudden dyspnea. Although their initial clinical mani-festations may be very similar, the potential developmentof hypovolemic shock leading to rapid clinical deteriora-tion distinguishes SHT from PSP, as can be observed inour case as well as in many other case reports [6, 9–12].A study by Kakaris et al. pointed out that, among 71patients diagnosed with SHT, 29.5% presented hemody-namic instability upon admission [3]. Before this urgencycharacteristic, we highlight the importance of maintaining

Figure 1: Chest X-ray revealed left pneumothorax with niveau level.

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a low threshold suspicion for this diagnostic possibility inpatients with compatible profile who present with charac-teristic symptoms.

Upright CXR is a routinely diagnostic tool by demon-strating typical radiological evidence of pneumothoraxassociated with an air-fluid level, even though up to 10%of cases may present only with pneumothorax on a firstmoment due to the early execution of this diagnostic testingor to the contention of the bleeding by a pleural adhesion.CT is normally not necessary, but it may be helpful if thediagnosis remains uncertain or to exclude secondary causesof hemothorax [1, 7].

Once diagnosticated, fluid reposition and chest tubeinsertion must be promptly provided in order to allow pul-monary reexpansion and stabilize the patient. Posteriorly,ultimate treatment decisions must be individualized basedon each patient’s clinical condition [8, 9].

As surgery indication criteria and the ideal timing forsuch intervention remain unresolved questions in the litera-ture, there seems to be a trend increasingly favorable towardsearly surgical intervention in all patients diagnosed with SHTconsidering the potential for this condition to lead to suddenclinical deterioration and the risks of retained blood clotswithin the pleural cavity [8, 12].

A retrospective study by Hsu et al. reported that 87.6%out of 201 patients with SHT required surgical interven-tion after closed tube thoracostomy [4]. In another studyby Chang et al., up to 30% of patients initially managedconservatively needed posterior operations or presented

Figure 2: Noncontrast chest CT scan showed hydropneumothorax with left lung collapse and mediastinum deviation.

Figure 3: Chest X-ray revealed satisfactory pulmonary expansionafter thoracic tube insertion.

Figure 4: Thoracoscopic visualization of the bleeding source on thetop of the left pleural cavity after hemostasis (∗) and region ofbullectomy (+).

3Case Reports in Pulmonology

prolonged hospital stay owing to later complications such asempyema and persistent air leakage [13]. Surgical strategiesfor approaching SHT include open thoracotomy or VATS.While the first is the preferred modality on the emergencysetting in patients who present with active bleeding andhemodynamic instability, the latter may be performed elec-tively after the clinical condition has stabilized [7].

There are many successful experiences with VATS inthe treatment of SHT currently reported [14]. Shorter hos-pital stay, less blood loss, and decreased need for bloodtransfusion are mentioned by Chang et al. as advantagesexperienced by patients who underwent early VATS com-pared to conservative treatment alone [13]. Moreover, lesserinvasive techniques have been refined to optimize thesebenefits, as attested by a recent report in which a successfuluniportal VATS was performed for the management ofSHT [10]. In our case, due to persistent blood drainage bychest tube, the patient underwent elective VATS with abra-sive pleurodesis and bullectomy, obtaining satisfactoryresults and fast recovery.

On the other hand, some authors defend that conser-vative work-up may be sufficient in selected cases. Hacii-brahimoglu et al. advocate that thoracic drainage alone issufficient in most cases, and surgical intervention is expend-able if the blood outflow from the drainage tube has stoppedin less than 24 hours [5]. A study by Kakaris et al. conductedonly conservative treatment in 16 out of 71 patients withSHT [3].

The occurrence of contralateral pneumothorax followingan SHT episode is not commonly observed in the literature,as we can see in a review that analyzed 8 case series, contain-ing 201 patients with SHT, in which no recurrence of pneu-mothorax was observed in a follow-up period that rangedfrom 5 months to 8 years [4]. On a prospective study byKim et al., 17 patients were diagnosed with SHT, of which 2of them evolved with contralateral spontaneous pneumo-thrax after being initially treated with thoracostomy [8].Kakamad et al. (2016) reports the case of a patient withSHT who was initially treated with thoracotomy, and afterone month was readmitted with contralateral pneumothorax[9]. Our patient presented asymptomatic right pneumotho-rax one week after being submitted to VATS. To our knowl-edge, this time interval of recurrence was the earliest amongthe cases described in the literature.

4. Conclusion

Therefore, clinicians acting on emergency departmentsmust be aware of SHT as a diagnostic possibility in youngmale patients presenting with abrupt onset of hypovolemiasigns with no apparent cause, despite being an unusualcondition [1].

Currently, predominant opinion supports that all casesof STH should undergo early surgical intervention, butsuch belief is mostly based on different center case series.Thus, we conclude that additional prospective, randomized,and multicentric trials are warranted to define consistenteligibility or exclusion criteria for conservative treatmentas well as to design standardized management algorithmsfor this entity.

Conflicts of Interest

The authors declare that there is no conflict of interestregarding the publication of this article.

Figure 6: Noncontrast chest CT scan demonstrates the presence ofpneumothorax on the right hemithorax.

Figure 5: Anatomic specimen operatively resected showing a 0.3 cmdiameter bullae in the subpleural space (arrow).

4 Case Reports in Pulmonology

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